Clinical Practice

Primary Malignant Melanoma of Maxillary

Gingiva — A Case Report and Review
of the Literature
Contact Author
Ajit Auluck, BDS, MDS; Lewei Zhang, BDS, Dip Oral Path, PhD, FRCD(C);
Dr. Auluck
Rajeev Desai, MDS; Miriam P. Rosin, PhD Email: drajitauluck@
gmail.com

ABSTRACT

Dentists may encounter pigmented lesions in routine clinical practice. In most cases, the
lesions are asymptomatic and benign in nature; however, rarely, a pigmented lesion
can be a sign of malignancy. We report a case of malignant melanoma of the maxillary
gingiva to highlight the importance of biopsy and periodic follow-up of patients with
unusual focal pigmented lesions in the oral cavity.

For citation purposes, the electronic version is the definitive version of this article: www.cda-adc.ca/jcda/vol-74/issue-4/367.html

T
he oral cavity is a common site for various pigmentation was a normal variant for her
pigmented lesions: amalgam tattoo, mel- ethnic group. The patient remained asymp-
anotic macule, smoker’s melanosis, racial tomatic until 2 weeks before her visit to the
pigmentation, nevus, drug-induced pigmenta- clinic when she noticed “swelling” of her gums
tion, systemic diseases and, of course, mel- and some discomfort in chewing.
anoma. Although most pigmented lesions The patient was moderately built and
are benign and of no clinical consequence, nourished, and her vital signs were within
clinicians must differentiate between the large normal limits. Intraoral examination showed
number of benign lesions and the rare serious a diffuse lesion extending from the right
diseases, most notably melanoma. We report maxillary canine to the left maxillary ca-
a case of misdiagnosed malignant melanoma nine region. On the labial side, the lesion was
and briefly review the differential diagnosis of slightly elevated and displayed red-black dis-
oral pigmented lesions. coloration (Fig. 1); on the palatal side, al-
though the lesion was only slightly elevated
Case Report between teeth 13 and 12, it was markedly exo-
A 32-year-old Asian woman reported for phytic between teeth 11 and 23 (about 1.5 cm
consultation regarding a rapidly growing pig- in diameter) and the mass had an irregular
mented mass in the maxillary anterior region. border and a rough surface (Fig. 2). The cen-
Three months earlier, she had developed an tral incisors within the lesion were mobile
asymptomatic bluish-black patch on her an- (grade 2). On palpation, there was no tender-
terior maxillary gingiva. Esthetic concerns ness, bleeding or regional lymphadenopathy.
prompted her to visit her dentist, who ruled An intraoral radiograph could not be ob-
out any pathology and suggested that this tained because of difficulty in film placement.

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Although panorex has limited value in

determining bone changes in anterior
teeth, a panoramic radiograph was taken
to evaluate possible marked bone de-
struction. No apparent abnormality was
seen (Fig. 3). An incisional biopsy was
performed under local anesthesia.
Hematoxylin- and eosin-stained sec-
tions of the biopsy specimen showed
numerous atypical melanocytes at
Figure 1: Intraoral photograph showing Figure 2: Intraoral photograph
the epithelial connective tissue junc-
bluish-black pigmentation on the labial showing a black proliferative growth tion, proliferating laterally along the
attached gingiva extending from the right on the palatal aspect of the anterior basal cell layer and infiltrating verti-
maxillary canine region to the central teeth. cally into the deeper connective tissue
incisor, crossing the midline and extending
to the left lateral incisor.
(Fig. 4). The malignant melanocytes
were pleomorphic and hyperchromatic
and contained brownish-black granular
pigment in the cytoplasm. The tumour
cells stained intensely with HMB-45,
an antibody to melanoma antigen, con-
firming the melanocytic nature of the
lesion (Fig. 5). The lesion was diagnosed
pathologically as malignant melanoma.

Discussion
Melanoma is a malignant neoplasm
Figure 3: The panoramic radiograph did not reveal any abnormality. arising from the neural crest cells.
During embryologic development, mel-
anocytes migrate from the neural crest
a b
into the epithelial lining of the skin
and, in the developed skin, they reside
primarily in the basal epithelial layer.1
Because the oral cavity develops from an
ectodermal depression or invagination,
the epithelial lining of the oral mucosa,
similar to skin, normally contains mel-
anocytes in its basal layer,1–3 which can
evolve into melanoma as in the skin.
Figure 4: Photomicrographs showing infiltration of malignant melanocytes into the
connective tissue (hematoxylin and eosin stain). Original magnification: a. ×10; b. ×40.
Malignant melanoma is a deadly
disease. Although it constitutes only
a
3%–5% of all cutaneous malignan-
b
cies, it accounts for most skin cancer-
related deaths (77%).4 Oral melanoma is
extremely rare and accounts for less than
1% of all melanomas1,5,6 and 1.6% of all
head and neck malignancies.
Despite the rarity of the disease, mel-
anoma is the most important pigmented
lesion in the oral cavity because of its
deadly nature and most, if not all, oral
Figure 5: Photomicrographs showing HMB-45 positive malignant melanocytes
(immunoperoxidase stain). Original magnification: a. ×10; b. ×40.
biopsies of pigmented lesions are aimed
at excluding malignant melanoma. It
is well known that early diagnosis and
treatment of melanoma can reduce mor-
tality rate. If diagnosed early, when the

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ease of adults (usually > 40 years

Bluish-black discoloration of oral mucosa of age) and rare in people < 20
years; the female-to-male ratio is
Usually due to metals/melanin pigment
3:1.8 It can afflict any ethnic group,
although the Japanese popula-
tion appears to be more suscept-
Localized pigmentation Diffuse pigmentation
ible.9,10 However, melanoma does
have a predilection for certain oral
Exclude pigmentation due to metals sites — the hard palate and max-
Physiological pigmentation
like amalgam, graphite, lead, etc. illary gingiva,4,11–13 as in this case.
Present from birth
In the oral cavity, amalgam tattoos usually Therefore, dentists should pay par-
occur near restorations ticular attention to pigmented le-
Melanotic macule Smoker’s melanosis sions in these regions.
History of smoking Clinical presentations of mel-
Small size, mostly on lips, increase in
melanin synthesis anoma vary tremendously. It
Endocrine disorders like Addison’s
appears most commonly as a pig-
Nevus mented lesion varying from dark
disease/Cushing’s syndrome
Increased proliferation of melanocytes,
Look for systemic signs and symptoms
brown to blue-black, although
usually from birth
some can be amelanotic.14 In the
latter case, it is not possible to make
Malignant melanoma HIV-associated melanosis
a clinical diagnosis of melanoma;
Dark, irregular borders, asymmetric Advise ELISA for HIV
and rapid growth
only a biopsy can reveal the true
nature of the lesion. A melanoma
Postinflammatory pigmentation Associated with such syndromes lesion can be flat (macule) or ele-
due to healing of lesions such as as Albright’s and Peutz Jegher’s vated (nodule or tumour), with or
lichen planus
General examination to find without ulceration or an erythema-
other associated features tous border, and it can be small or
large. However, dentists should be
Figure 6: Differential diagnosis of pigmented lesions in the mouth, including characteristic
highly suspicious of malignant mel-
features that help in diagnosis. ELISA = enzyme-linked immunosorbent assay. Note: Biopsy must anoma in the following situations
be advised if there is an increase in size, a change in colour or any proliferative changes associ- (Box 1): variegation in colour
ated with a pigmented lesion. (red to black-brown) within a pig-
mented lesion, particularly when
malignant cells are limited to the epidermis or invasion it has an asymmetrical or irregular outline; sudden ap-
is minimal, melanoma is either 100% curable by excision pearance of a large pigmented lesion, particularly when
(for in situ lesion) or is associated with a 5-year survival it has an exophytic component, as in this case, or has
rate of 95% (for lesions < 1 mm in thickness without erythematous or ulcerated areas. It should be noted that
ulceration).7 In contrast, the 5-year survival rate for even when melanoma becomes exophytic or ulcerated,
cutaneous melanomas > 4 mm thick with ulceration is it generally lacks the induration and rolled ulcerated
only 45%. border frequently seen in oral squamous cell carcinoma,
For melanoma in the oral cavity, the prognosis is the most common oral cancer. These features should not
much worse: the 5-year survival rate is generally in the be used to judge whether a pigmented lesion is malig-
range of 10% to 25%, partly because detection is more nant, as the radial growth phase of melanoma could be
difficult as pigmented lesions in the oral cavity are less prolonged with minimal or no invasion and, during this
visible than on the skin. For this reason, the dental pro- phase, atypical melanocytes exhibit a pagetoid (upward
fession plays an important role in the early diagnosis of migration) mode of spread resulting in uniform epithelial
oral melanoma. In our case, if the patient had been diag- thickening1 and a lack of focal indurations.
nosed immediately, when the lesion was flat and prob-
ably limited to the epithelium, the prognosis would have Differential Diagnosis of Large Pigmented Lesions
been much better; the 3-month delay resulted in > 4-mm Many melanomas grow rapidly; dentists should be
thickness of the lesion and ulceration. particularly cautious when confronted with a large pig-
Differentiation of melanoma from various oral pig- mented lesion, as in this case. Racial pigmentation, drug-
mented lesions can be a daunting task (Fig. 6) There are induced pigmentation, smoker’s melanosis and some
no striking features to distinguish oral melanoma from syndromes can all present as a large area of pigmentation
many other oral pigmented lesions. It is generally a dis- in the oral region.

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Box 1 ABCDE warning signs suggestive of early melanoma flammatory pigmentation should be related to a previous
condition, such as long-standing erosive lichen planus at
Asymmetry: The shape of the lesion is not the same on the site.16 Rarely, systemic diseases, such as Addison’s dis-
both sides. ease, can present diffuse pigmentation, but a careful med-
Border irregularity: The edges are ragged, notched or ical history should help to establish these conditions.
blurred.
All of these conditions warrant a biopsy if a diagnosis
of melanoma cannot be firmly excluded.
C olour variation: Pigmentation is not uniform and may
display shades of tan, brown or black. White, reddish or Differential Diagnosis of Small Pigmented Lesions
blue discoloration is of particular concern. The most common oral pigmentations are a result of
Diameter: A diameter greater than 6 mm is characteristic amalgam tattoo, melanotic macules and graphic tattoo
of melanoma, although some may have smaller diam- (from pencil lead). These lesions are generally small
eters. Any growth in a nevus warrants an evaluation. (< 1 cm, although, in rare situations, amalgam tattoo
Evolving: Changes in the lesion over time are character- can be large) and flat, grey-black to blue-black in colour
istic. This factor is critical for nodular or amelanotic (amalgam and graphic tattoos) or light to dark brown
(nonpigmented) melanoma, which may not exhibit the (melanotic macule). Occasionally, nevi also occur in the
classic criteria listed above. oral cavity as pigmented lesions and tend to be < 1 cm in
diameter, although they can be either flat or elevated. Nevi
vary in colour from amelanotic, to brown to blue-black.
This case should not have been confused with racial For small pigmented macules, the most likely diag-
pigmentation, which is a physiological condition, for a nosis is one of the above entities, particularly amalgam
number of reasons. First, racial pigmentation is more tattoo. Unfortunately, in rare cases, a small flat pigmented
diffuse and, on gingiva, it will be generalized rather macule can represent an early stage of a malignant mel-
than confined to one quadrant as in this case. Second, anoma. Cases of melanoma have been reported to be
preceded by an asymptomatic flat pigmented macule for
racial pigmentation will be present from an early age;
periods of several months to 5 years17 before they start to
the sudden appearance of a pigmented lesion, as in this
show the symptoms and signs of malignant melanoma,
case, should immediately preclude the diagnosis of ra-
such as sudden growth, a change in colour or becoming
cial pigmentation. Duration of pigmentation is always
exophytic and hemorrhagic. In this case, the elevated
critical in the differential diagnosis of oral melanoma.
lesion was preceded 3 months earlier by a pigmented
The rule of thumb is that if a pigmented area has not
macule. In fact, approximately a third of melanomas are
changed in 5 years, it cannot be melanoma and no biopsy
preceded by asymptomatic pigmented macules.1 Thus,
is required. Third, racial pigmentation will always be flat
a biopsy of a benign-looking small pigmented lesion is
and asymptomatic; the rapid development of elevated
warranted to rule out early melanoma, unless lesions
areas and symptoms in this case, again, strongly point
have been present for a long time without change or the
to a diagnosis of melanoma. Unfortunately, many mel-
diagnosis of amalgam tattoo can be established firmly by
anomas frequently remain asymptomatic and are not
radiographic demonstration of amalgam speckles or reli-
diagnosed until there is a breakdown of the epithelium able history.
or hemorrhage.15 Surgery is believed to be the most effective treat-
Drug-induced pigmentation tends to be extensive and ment for melanoma. For cutaneous melanoma, wide re-
has a predilection for the hard palate. However, the ap- section with surgical margins of 2.5 cm is necessary.18
pearance of such a pigmented area should be associated Immunotherapy with interferon may also be beneficial.18
with the intake of a medication, particularly medications If there is disseminated metastatic disease, chemotherapy
known to cause pigmentation. In the absence of any and radiation therapy should be advised.19 Dentists must
medication, melanoma should be suspected when a large coordinate with oral and maxillofacial surgeons, medical
pigmented area is present on the hard palate. Even in the oncologists and radiation therapists to ensure proper
case of a history of medication, if there is any doubt, a management of cases of malignant melanoma.
biopsy should be performed.
Smoker’s melanosis or postinflammatory pigmenta- Conclusions
tion may present as large pigmented areas, but this pig- The oral cavity is a common site for pigmented le-
mentation tends to be brownish in colour and generally sions, most of them benign. Dentists should keep the
not as dark as melanoma. Smoker’s melanosis is seen in possibility of malignant melanoma in mind during any
people with a long habit of smoking and is frequently differential diagnosis of a pigmented lesion. When in
located on the mandibular anterior gingiva (in cigarette doubt, the dentist should refer the patient to a specialist
smokers) or the buccal mucosa (in pipe smokers). Postin- or perform a biopsy. a

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